Summary
A 68-year-old man presented with general fatigue, increasing adynamia, weakness, vertigo and recurrent syncope. Six weeks earlier the diagnosis of a macroprolactinoma had been established based on a greatly elevated prolactin concentration (161 170 μU/l) and MR-evidence of a 3.5 cm measuring pituitary mass. The patient had been started on cabergoline (1.5 mg weekly). Orthostatic hypotension due to the dopamine agonist was considered very likely and carbergoline therapy was stopped. However, there was no relief of the symptoms and further syncopes followed. Testing of blood pressure and heart rate regulation, selective testing of postganglionic cardiac neurons with [123J] metaiodobenzylguanidine scintigraphy provided evidence of grossly impaired neurogenic cardiovascular regulation due to failure of postganglionic efferent sympathetic activity. This is characteristic for pure autonomic failure. The patient was treated symptomatically with high fluid intake, compression stockings, fludrohydrocortisone (0.1 mg o.d.s.), piroxicam (20 mg o.d.s.) and etilephrin (10 mg q.d.s.), which enabled him to cope with daily activities without syncope. This case shows that vertigo in a patient with macroprolactinoma is not always related to drug therapy but may be related to other causes.
Key words:
Orthostatic hypotension - autonomic failure - prolactinoma
References
-
1
American Autonomic Society and American Academy of Neurology .
Consensus statement on the definition of orthostatic hypotension, pure autonomic failure and multiple system atrophy.
Clin Autonomic Res.
1996;
6
125-126
-
2
Arai K, Kato N, Kashiwado K, Hattori T.
Pure autonomic failure in association with human alpha-synucleinopathy.
Neuroscience Letters.
296 (2-3)
171-173
2000;
-
3
Becker K, Keuser R, Weber M, Steffen H M.
Autonomic failure as the cause of vertigo and prolactinoma.
Med Klin.
90 (7)
398-402
1995;
-
4
Bernard A, Koch J, Vetter W.
Prevalence of hypotension and symptomatic, orthostatic dysregulation in a patient sample of a medical polyclinic.
Schweiz Rundsch Med Prax.
88 (13)
547-558
1999;
-
5
Colao A, Annunziato L, Lombardi G.
Treatment of prolactinoma.
Ann Med.
30 (5)
452-459
1998;
-
6
Dae M W, O'Connell J W, Botvinick E H.
Scintigraphic assessment of regional cardiac adrenergic innervation.
Circulation.
79 (3)
534-644
1989;
-
7
Esler M, Jackman G, Kelleher D, Skewa H, Jennings G, Bobik A, Korner P.
Norepinephrine kinetics in patients with idiopathic autonomic insufficiency.
Circ Res.
1980;
46
I47-I48
-
8
Gasnier B, Roisin M P, Scherman D.
Uptake of meta-iodobenzylguanidine by bovine chromaffin granule membranes.
Mol Pharmacol.
29 (3)
275-280
1986;
-
9
Glowniak J V, Kilty J E, Amara S G.
Evaluation of metaiodobenzylguanidine uptake by the norepinephrine, dopamine, and serotonin transporters.
J Nucl Med.
1993;
34
114-116
-
10
Grubb B P, Klingenleben T.
Syndromes of autonomic insufficiency associated with orthostatic intolerance: classification, diagnostic and therapeutic approach.
Z Kardiol.
1999;
88
541-549
-
11
Hague K, Lento P, Morgello S, Caro S, Kaufmann H.
The distribution of Lewy bodies in pure autonomic failure: autopsy findings and review of the literature.
Acta Neuropathol.
1997;
94
192-196
-
12
Hale W A, Chamblis M L.
Should primary care patients be screened for orthostatic hypotension?.
J Fam Pract.
48 (7)
547-552
1999;
-
13
Jaques S, Tobes M C, Sisson J C.
Sodium dependency of uptake of norepinephrine and m-iodobenzylguanidine into cultured human pheochromocytoma cells: evidence of uptake-one.
Cancer Res.
1987;
47
3920-3928
-
14
Jaques S, Tobes M C.
Comparison of the secretory mechanisms of meta-iodobenzylguanidine (MIBG) and norepinephrine (NE) from the cultured bovine adrenomedullary cells.
J Nucl Med.
1985;
26
17
-
15
Kreiner G, Wolzt M, Fasching P.
Myocardial m[123I]iodobenzylguanidine scintigraphy for the assessment of adrenergic cardiac innervation in patients with IDDM: Comparison with cardiovascular reflex tests and relationship to left ventricular function.
Diabetes.
44 (5)
543-549
1995;
-
16
Langer A, Freeman M R, Josse R G.
Metaiodobenzylguanidine imaging in diabetes mellitus: assessment of cardiac sympathetic denervation and its relation to autonomic dysfunction and silent myocardial ischemia.
J Am Coll Cardiol.
25 (3)
610-618
1995;
-
17
Mathias C J, Mallipeddi R, Bleasdale-Barr K.
Symptoms associated with orthostatic hypotension in pure autonomic failure and in multiple system atrophy.
J Neurol.
1999;
246
893-898
-
18
Mathias C J.
Orthostatic hypotension: Causes, mechanism, and influencing factors.
Neurology.
1995;
45
S6-S11
-
19 Matthews M R. Assessing the peripheral ganglia in autonomic failure. In: Bannister R (ed). Autonomic failure, Second Edition Oxford Medical Publications, Oxford 1988: 521-543
-
20
Meredith I T, Eisenhofer G, Lambert G W, Jennings G L, Thompson J, Esler M D.
Plasma norepinephrine responses to head-up tilt are misleading in autonomic failure.
Hypertension.
1992;
19
628-633
-
21 Oppenheimer D. Neuropathology of autonomic failure. In: Bannister R (ed). Autonomic Failure, Second Edition Oxford Medical Publications, Oxford 1988: 451-463
-
22
Polinsky R J, Nee L E.
Autonomic failure: Nosology, clinical evolution, and prognosis.
Ann Neurol.
1989;
26
121
-
23
Polinsky R J, Kopin I J, Ebert M H, Weise V.
Pharmacological distinction of different orthostatic hypotension syndromes.
Neurology.
1981;
31
1-7
-
24
Robertson D, Robertson R M.
Causes of chronic orthostatic hypotension.
Arch Int Med.
1994;
154
1620-1624
-
25
Schnell O, Kirsch C M, Stemplinger J.
Scintigraphic evidence for cardiac sympathetic dysinnervation in long-term IDDM patients with and without ECG-based autonomic neuropathy.
Diabetologica.
38 (11)
1345-1352
1995;
-
26
Silke B, Spiers J P, Boyd S, Graham E, McParland G, Scott M E.
Evaluation of non-invasive blood pressure measurement by the Finapres method at rest and during dynamic exercise in subjects with cardiovascular insufficiency.
Clin Autonomic Res.
1994;
4
49-56
-
27
Sisson J C, Wieland D M, Sherman P.
Metaiodobenzylguanidine as an index of the adrenergic nervous system integrity and function.
J Nucl Med.
28 (10)
1620-1624
1987;
-
28
Sisson J C, Shapiro B, Meyers L.
Metaiodobenzylguanidine to map scintigraphically the adrenergic nervous system in man.
J Nucl Med.
28 (10)
1625-1636
1987;
-
29
Sisson J C, Lynch J J, Johnson J.
Scintigraphic detection of regional disruption of adrenergic neurons in the heart.
Am Heart J.
1988;
116
67-76
-
30
Van Ingelghem E, Van Zandijcke M, Lammens M.
Pure autonomic failure: a new case with clinical, biochemical, and necropsy data.
J Neurol Neurosurg Psychiatry.
1994;
57
745-747
-
31
Wieland D M, Mangner T J, Inbasekaran M N.
Adrenal medulla imaging agents: a structure-distribution relationship study of radiolabeled aralkylguanidines.
J Med Chem.
1984;
27
149-155
-
32
Ziegler M G, Lake C R, Kopin I J.
The sympathetic-nervous-system defect in primary orthostatic hypotension.
N Engl J Med.
1977;
296
293-297
Dr. Lysann Seiler
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